Chronic recurrent multifocal osteomyelitis: report of five cases

Chronic recurrent multifocal osteomyelitis is a rare disease of unknown etiology that mainly affects children. We present a retrospective review of the medical records of five patients with a diagnosis of chronic recurrent multifocal osteomyelitis in an infectious diseases clinic at a tertiary center. We describe the epidemiological, clinical and radiological features, laboratory, microbiological and histological findings, treatment, course and prognosis in these patients. There is no specific treatment for chronic recurrent multifocal osteomyelitis. There are new treatment options such the combination of azithromycin and calcitonin in children without clinical improvement with non-steroidal anti-inflammatory drugs.     

Actinomycosis presenting as osteomyelitis in the pediatric population

Osteomyelitis attributable to Actinomyces often results in recurrent disease and inconsistent responses to antimicrobial agents. We present data for 4 patients and a review of the 15 previously described pediatric cases of actinomycosis presenting as osteomyelitis. Fourteen cases involved the mandible and 5 cases involved other sites. All mandibular cases required at least 1 debridement, with 4 of the cases requiring multiple debridements.     

Clinical presentation and management of Pseudomonas osteomyelitis

To determine the incidence and clinical characteristics of Pseudomonas aeruginosa osteomyelitis in children, the records of 144 hospitalized patients under 19 years of age were reviewed; 104 fulfilled the study criteria for the diagnosis of acute or chronic osteomyelitis. Pseudomonas aeruginosa was recovered from 10.6 percent of the children and was the second most common pathogen isolated. In comparison to children with staphylococcal infections, patients with pseudomonal osteomyelitis were significantly older, gave an antecedent history of penetrating trauma, and lacked clinical and laboratory evidence of systemic illness. The data collected in this study suggest that osteomyelitis due to Pseudomonas aeruginosa is a distinct entity with clinical features differing from those of Staphylococcus aureus. Management should be directed at adequate surgical debridement followed by 10 to 21 days of antimicrobial therapy.     

Successful management of chronic multifocal Q fever Osteomyelitis with adjuvant interferon-gamma therapy

We present a 3-year-old girl who had chronic recurrent multifocal osteomyelitis caused by Coxiella burnetii despite long-term dual antibiotic therapy. Excellent clinical response was achieved and sustained when immunomodulatory therapy with interferon-γ was initiated. This is the case of a first child who was successfully treated with interferon-γ as adjuvant therapy for chronic multifocal Q fever osteomyelitis.     

Chronic recurrent multifocal osteomyelitis of the spine and mandible: case report and review of the literature

At initial presentation, chronic recurrent multifocal osteomyelitis may mimic acute hematogenous osteomyelitis; however, cultures of affected bone are sterile. Nuclear scintigraphy identifies additional foci of involvement that present concurrently or sequentially. Unlike acute bacterial osteomyelitis, chronic recurrent multifocal osteomyelitis seems unaffected by antibiotic therapy and typically responds to treatment with antiinflammatory drugs. Surgical decortication has been reported for refractory cases. The case presented here illustrates the rare involvement of the mandible after initial presentation in the spine of a 4-year-old girl and the refractory nature of the disease over 6 years despite treatment with various medical and surgical therapies.     

The syndrome of chronic recurrent multifocal osteomyelitis and congenital dyserythropoietic anaemia. Report of a new family and a review

A new autosomal recessive syndrome of chronic recurrent multifocal osteomyelitis (CRMO) and congenital dyserythropoietic anaemia (CDA) with microcytosis has recently been described in four children (two sibships) of one consangineous Arab family. In this report, we describe the clinical features and course of the syndrome of CRMO and CDA in two additional patients (one sibship) from another consanguineous Arab family and review the literature. The two patients (brother and sister), the products of a consanguineous marriage, developed the syndrome at an early age of 3 weeks and 2 months respectively. The diagnosis of CRMO was confirmed by radiological and technetium isotope bone scans. Bone marrow studies confirmed the diagnosis of CDA. Peripheral blood films showed hypochromia and microcytosis. The sites involved by CRMO were periarticular, mainly around the elbow, knee, wrist and small joints of the hand. The brother is now 21 years old and the sister 3.5 years old and CRMO is still active with frequent relapses. The brother developed flexion deformities at the age of 13 years. Both patients failed to thrive; weight and height were below the 5th percentile. CONCLUSION: This is the second report of the syndrome of chronic recurrent multifocal osteomyelitis and microcytic congenital dyserythropoietic anaemia, confirming it as a clinical entity, inherited as an autosomal recessive trait. The disease is characterised by an early onset, long clinical course of remissions and relapses, and seems to be different from the sporadic form of chronic recurrent multifocal osteomyelitis.     

儿童慢性骨髓炎应用抗感染活性骨Ⅰ期植骨治疗的疗效评价

目的 验证抗感染活性骨(anti-infective reconstituted bone xenograft,ARBX)Ⅰ期植骨治疗儿童慢性骨髓炎的疗效.方法 自2001年9月对21例慢性骨髓炎患儿进行病灶清除,应用抗感染活性异种骨(anti-infective reconstituted bone xenograft,ARBX)Ⅰ期植骨治疗,其中获得12个月以上随访的儿童慢性骨髓炎患儿共16例.包括血源性慢性骨髓炎8例,原发性慢性骨髓炎3例,创伤性慢性骨髓炎5例.结果 16例中除1例骨髓炎复发,1例骨髓炎合并大段骨缺损在术后发生骨不连外,其余14例感染均获得控制无复发,病变部位均获得确实骨修复.本组感染治愈率(感染控制且无复发)为93.8%,完全治愈率(感染控制既无复发又完成骨修复)为87.5%.结论 ARBX具有高效诱导成骨活性和强效抗感染能力,是Ⅰ期植骨治疗儿童慢性骨髓炎的有效方法.     

Burkholderia gladioli osteomyelitis in association with chronic granulomatous disease: case report and review

We describe a case of insidious small bone osteomyelitis and soft tissue abscess with Burkholderia gladioli in a 6-year-old Caucasian boy with chronic granulomatous disease. DNA sequencing of the 16S ribosomal RNA gene confirmed the bacterial identification. Clinical cure was achieved with a combination of antimicrobial therapy and surgical debridement. A review of infections caused by Burkholderia spp., other than Burkholderia cepacia complex, in pediatric patients with chronic granulomatous disease is provided.     

Rare forms of chronic osteomyelitis in children (author's transl)

4 cases of chronic osteomyelitis in children and adolescents are described. The first one concerns a girl with severe plasmacellular osteomyelitis of the right upperarm. As the treatment with antibiotics failed a partial resection of the right humerus, followed by plastic covering of the resulting defect was successful. Two further cases of chronic metatarsal osteomyelitis of both feet could be classified as a kind of "sclerosing osteomyelitis Garré", mostly corresponding to the clinical feature of the "chronic recurrent multifocal osteomyelitis" (Bj?rkstén et al., 1978). Furthermore, a case of chronic ostitis localized in the symphyseal area is described, the clinical symptoms of which were resembling to the cortical osteoid, a special kind of sclerosing osteomyelitis. It is suggested that these different forms of chronic osteomyelitis in children are caused by bacterial infections. Nevertheless, this hypothesis remains to be proved. The problems of the differential diagnosis are discussed. Generally, the only therapy with antibotics is not successful, whereas the combination of removing of the inflammatory process with plastic covering of the bone-defect usually results in recovery.     

Bacterial bone and joint infections in childhood--a review. 4. Subacute and primary-chronic osteomyelitis, rare forms of osteomyelitis

This is an overview of the most important aspects of pathogenesis, etiology, diagnostics, therapy and differential diagnostics of the subacute and primary chronic osteomyelitis in children. This group of disease includes Brodie's abscess, plasma cellular osteomyelitis, sclerosing osteomyelitis (Garré) and the chronic recurrent multifocal osteomyelitis. The treatment of children with these not completely understood diseases requires a close cooperation between pediatricians, pediatric surgeons and radiologists.     

Chronic recurrent osteomyelitis with clavicular involvement in children: diagnostic value of different imaging techniques and therapy with non-steroidal anti-inflammatory drugs

Chronic recurrent, uni- or multifocal osteomyelitis (CRMO), an inflammatory disorder of unknown origin, involves mk:/night/arul/4310946m.3dultiple osseous sites and may affect the clavicle. We report on 6 children with clavicular involvement out of 11 children suffering from CRMO. The major clinical symptoms were local swelling and pain. Five children had hyperostosis of the clavicle and synovitis of adjacent joints. Histology showed chronic osteomyelitis with a predominance of lymphocytes in the inflammatory infiltrates. Cultures of biopsy tissue specimens were sterile. The patients were followed for at least 3.5 years. Three patients had up to six relapses. The most effective diagnostic tools to define CRMO were standard X-ray and bone scan in combination with biopsy and cultures. In our patients CT and MRI were misleading as they suggested the presence of malignancy. However, the sensitivity of MRI to detect involvement of bone, adjacent joints and soft tissues were better in comparison to X-ray or bone scan. Non-steroidal anti-inflammatory drugs were effective in reducing pain, swelling and limitation of motion. Reconstructive surgery was not indicated in any case. The long-term outcome of growth and function of affected bones was excellent. Conclusion Diagnosis of chronic osteomyelitis of the clavicle should be made by history and physical examination and be confirmed by standard X-ray, bone scan and open biopsy. In contrast MRI and CT can provide data on the involvement of adjacent joints, soft tissue and muscles especially in the early process of disease, but do not add information relevant to the patient's management. Treatment with non-steroidal anti-inflammatory drugs is rapidly beneficial in most patients. Received: 24 January 1997 / Accepted: 26 May 1997     

Successful treatment of chronic recurrent multifocal osteomyelitis with tumor necrosis factor-alpha blockage

We describe the case of an 18-year-old girl with chronic recurrent multifocal osteomyelitis (CRMO) over a period of 10 years. She had suffered predominantly from very painful recurrent swelling of her cheeks. Various therapeutic regimens including nonsteroidal antiinflammatory drugs and steroids had shown only a partial or temporary response. Because tumor necrosis factor-alpha-blocking agents have been successfully applied in Crohn's-associated CRMO and the related SAPHO (synovitis, acne, pustulosis, hyperostosis, and osteitis) syndrome, tumor necrosis factor-alpha-blocking therapy with infliximab was initiated. Thereafter, apart from 1 mild episode, no additional recurrences were observed during 21 months of follow-up. Infliximab was well tolerated, and steroids were tapered off. Our observation indicates that infliximab may be an effective therapeutic option in CRMO.     

Systematic review of systemic antibiotic treatment for children with chronic and sub‐acute pyogenic osteomyelitis

Background: There is a lack of consensus on the most appropriate antibiotics to treat children with chronic or sub‐acute pyogenic bacterial osteomyelitis and on the optimal duration of antibiotic therapy. Aim: To review the published evidence on the duration of antibiotic therapy and outcomes in children with chronic and sub‐acute pyogenic bacterial osteomyelitis. Methods: Systematic review of the literature. Results: We found no randomised controlled trials comparing different antibiotic regimens or comparing duration of antibiotic treatment for chronic or sub‐acute osteomyelitis in children. We found 14 observational case series published between 1973 and 2008. Most children with chronic osteomyelitis received 4–6 weeks of parenteral antibiotics followed by oral antibiotics to a total duration of 3–6 months. Small observational studies suggest that a shorter duration of parenteral and oral antibiotics may be equally effective. Conclusion: There is no high level evidence on the optimal duration of parenteral and oral antibiotics for children with chronic or sub‐acute osteomyelitis. A large randomised controlled trial is needed comparing short course parenteral and oral antibiotics with longer antibiotic duration.     

Chronic recurrent multifocal osteomyelitis

Chronic recurrent multifocal osteomyelitis is a rare disorder of unknown etiology, characterized by multiple bone lesions and a variable clinical course. We present a 10 year old boy with chronic recurrent multifocal osteomyelitis who improved after treatment with naproxen.     

Crohn's-associated chronic recurrent multifocal osteomyelitis responsive to infliximab

We report a case of chronic recurrent clavicular osteomyelitis in association with Crohn disease. Steroid therapy resulted in partial remission; however, intractable shoulder pain and an enlarging clavicular mass subsequently recurred. Infliximab therapy resulted in significant improvement in the degree of bone pain and resolution of the large sclerotic clavicular lesion.     

Osteomyelitis in patient with sickle cell disease

From 1977 to 1989, 23 children with sickle cell disease were identified as having 21 episodes of acute and 3 episodes of chronic osteomyelitis, respectively. The responsible organisms were found in 17 cases: Salmonella (12 cases), coagulase-negative Staphylococcus (3 cases). Haemophilus influenzae (1 case), Escherichia coli (1 case). The mean age was 7 7/12 years. In 15 patients, osteomyelitis occurred in 1 bone; osteomyelitis of more than one bone was recorded in 9 cases. The most commonly affected bone was the femur (7 episodes); 5 episodes of hand-foot syndrome with osteomyelitis occurred in children in the first 2 years of life (mean age 16 months). Two patients had a Salmonella vertebral osteomyelitis. Incision and drainage were performed in 5 cases and bone aspiration in 9 cases. Etiologic agents were obtained with these two procedures in respectively 5 and 3 cases. Radionuclide scans were used in 7 episodes: uptake on bone scan was increased in 5 cases and normal in 2. In all cases, the outcome was satisfactory. Differentiation from acute bone infarcts in difficult. An extensive workup is required to confirm the diagnosis of infection: early scintigraphy, bone aspiration or surgical biopsy in patients with negative blood cultures should be performed. Until the results of cultures, the antimicrobial regimen chosen for initial therapy should be broad enough to treat the likely etiologic agents including Salmonella.     

Chronic recurrent multifocal osteomyelitis in children: report of 17 cases]

Chronic recurrent multifocal osteitis (OCRM) is a rare condition in children, of unknown aetiology, which may be misdiagnosed as osteomyelitis, arthritis or tumour. PATIENTS AND METHODS: We present a retrospective multicentric study of 17 patients (five boys and 12 girls) with an average follow-up of 7.5 years (six months-25 years). RESULTS: A spectrum of presenting features is possible, ranging from bone lesions alone to lesions combined with arthritis, palmoplantar pustulosis or psoriasis. The diagnosis was delayed from two weeks to five years. Roentgenographic evaluation was often normal at the beginning of the disease or showed nonspecific bone reactions. Radioisotope bone scans assisted in establishing the diagnosis and in identifying lesions that were initially clinically silent. Bone biopsies were performed in seven cases. Histopathological examination showed only mild inflammatory nonspecific changes. Microbiological cultures were always negative. Treatments were different according to the evolution of the disease and the hospital. There was no response to antibiotics in seven patients. The response to nonsteroidal anti-inflammatory agents and steroids was moderate and often transient. Salazopyrine and pamidronate treatment used in two patients allowed a durable remission. We lost sight of four patients, pain persisted in three in spite of treatment, it disappeared in two with treatment, mild pain persisted in five without treatment and remission occurred in three without treatment. CONCLUSION: This study clarifies the clinical and radiologic features of chronic recurrent multifocal osteomyelitis. The recognition of this rare entity is often delayed and difficulties in patient management sometimes emerge from its usual protracted course.     

Oral antibiotic therapy for skeletal infections of children. II. Therapy of osteomyelitis and suppurative arthritis

Antimicrobial regimens consisting of a brief initial period of parenteral therapy followed by oral therapy were investigated in infants and children with suppurative bone and joint disease. There were 30 patients with acute hematogenous disease (19 osteomyelitis; three osteoarthritis; eight arthritis) and five with subacute or chronic osteomyelitis. Disease was due to Staphylococcus aureus in 26, Hemophilus influenzae in five, streptococci in three, and S. aureus plus Streptococcus pyogenes in one patient. Pus was removed by surgical drainage or needle aspiration. Oral therapy was monitored by assay of antibiotic concentration and bactericidal activity in serum. Adjustments in dosage were made when necessary to assure a peak serum bactericidal titer of at least 1:8. One patient progressed to chronic osteomyelitis but all other patients with acute disease responded well. Oral therapy provides increased patient comfort and decreases the risk of nosocomial infection associated with prolonged intravenous therapy. It should be carried out only under carefully monitored conditions in hospital to assure compliance and adequacy of serum bactericidal activity.     

Physeal involvement in chronic recurrent multifocal osteomyelitis

Chronic recurrent multifocal osteomyelitis is a recently recognized disease characterized by remissions and exacerbations of multiple bone lesions which radiographically and pathologically have the appearance of hematogenous osteomyelitis. No consistent etiology can be identified, and antimicrobial agents seem to have no beneficial effect. A review of the appearances of imaging modalities and the clinical and pathologic manifestations is undertaken in seven cases of CRMO. No therapeutic regimen resulted in consistent clinical or radiographic improvement. We suggest the sequelae of this process are not as benign as previously reported.     

Helicobacter pylori infection in recurrent abdominal pain

BACKGROUND: Helicobacter pylori is associated with chronic gastritis and peptic ulcer in adults and in children. The purpose of the present study was to analyze the association of recurrent abdominal pain and H. pylori infection in children and to evaluate the efficacy of antimicrobial treatment in patients with evidence of infection. METHODS: The clinical and histopathologic findings in children who underwent diagnostic upper endoscopy for recurrent abdominal pain were analyzed retrospectively. Patients with evidence of infection with H. pylori were treated with a combination of omeprazole, amoxicillin, and clarithromycin. Efficacy of treatment was assessed using the 13C-urea-breath test. RESULTS: H. pylori was found in histopathologic sections of 29 (40%) of 73 patients undergoing diagnostic endoscopy for recurrent abdominal pain. Five children (17%) were of Swiss ethnic origin, and 24 (83%) were non-Swiss. All the infected patients had chronic gastritis and 4 (14%) had ulcerations in the duodenum. Treatment with omeprazole, amoxicillin, and clarithromycin resulted in eradication of the infection in all and in resolution of the clinical symptoms in 15 (80%) of 19 patients who had a follow-up examination. CONCLUSIONS: The presented data suggest that gastritis induced by H. pylori may be associated with recurrent abdominal pain and that in Switzerland infections with H. pylori primarily involve persons who are non-Swiss. A combined therapy results in eradication of the bacterium and in improvement of the clinical symptoms in a significant majority of the patients.